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Pilocytic astrocytoma (PA) is a slow growing brain tumor that tend to occur in young patients. Although PA is often a benign tumor, dissemination to leptomeninges has been described in the literature. We describe a sixteen-yearold boy who initially presented with paraplegia and progressed to severe neurological features. An intradural intramedullary spinal cord tumor was identified in medullar MRI, and diagnosis of PCA by a surgical biopsy of the lesion was performed. Subsequently, patient developed an intracranial hypertension syndrome and Craniospinal MRI revealed an intracranial and spinal leptomeningeal dissemination. The patient was treated with courses of cytotoxic consisting of carboplatin - vincristine-based chemotherapy. He died of disease two months later. Our case emphasizes the lack of benefit of chemotherapeutic agents in PA with diffuse leptomeningeal dissemination and review existing literature. [ABSTRACT FROM AUTHOR]